Losartan
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Lack of efficacy: 2 case reports A case report of two patients described a 20-month-old girl and a boy [exact age of boy not stated], who exhibited lack of efficacy during treatment with losartan for Loeys-Dietz syndrome (LDS) and associated ulcerative colitis or LDS, respectively [routes not stated; not all dosages stated]. Case 1: The girl developed chronic bloody diarrhoea and intermittent fever from the age of 13 months, eventually leading to failure to thrive. Based on the laboratory test findings and her peculiar phenotypic features, she was diagnosed with LDS, which was confirmed by the detection of a mutation of transforming growth factor beta receptor type 2 (TGFBR2) already described in LDS (p.R528C). Infectious causes of diarrhoea were excluded. She tested negative on chronic granulomatous disease testing and genetic testing for interleukin (IL)-10 receptor mutations. No alterations of the mesenteric vessels were noted. Based on the findings examinations, she was diagnosed with chronic active ulcerative colitis associated with LDS. She was treated with mesalazine and prednisone with good clinical response, but the disease relapsed on prednisone tapering. Therefore, ciclosporin and azathioprine were added to her treatment, which resulted only in a limited improvement. Therefore, ciclosporin was discontinued, and infliximab was added to azathioprine and prednisone. However, only modest clinical improvement was noted, while tapering of prednisone often led to clinical relapses that required an increase of prednisone and short courses of antibiotics (metronidazole and ciprofloxacin). After 1 year (13 infusions) infliximab was stopped. At the age of 4 years, she developed two perianal extra-sphincteric abscesses, which were treated with antibiotic therapy. At the age of 30 months, she was diagnosed with autoimmune thyroiditis and was started on levothyroxine sodium [levothyroxine]. Notably, at the age of 20 months, she had started receiving losartan 0.7 mg/kg for LDS and associated ulcerative colitis without improvement of the intestinal disease, which was still active at the age of 4 years and 6 months. Case 2: The boy was diagnosed with LDS based on typical phenotypic features. The diagnosis was confirmed by genetic analysis (de novo mutation of TGFBR1, p.G217R). Thereafter, he started receiving treatment with carvedilol and losartan. However, no improvement in LDS was noted with losartan therapy. From the age of 26 months, he developed chronic bloody diarrhoea, intermittent fever and failure to thrive, with hypergammaglobulinaemia, anaemia and elevated ESR. Infectious causes of colitis were ruled out. He tested negative on chronic granulomatous disease testing and genetic testing for interleukin (IL)-10 receptor mutations. Based on the findings of colonoscopy, he was considered to have ulcerative colitis associated with LDS and was started on unspecified corticosteroids with good clinical response. But, the disease relapsed after corticosteroid discontinuation. Therefore, he received a second short co
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